A 16-year-old female patient with headache was admitted to our hospital. Radiological examination showed a Spetzler-Martin Grade III arteriovenous malformation (AVM) located at the left frontal lobe. Volume-staged stereotactic radiosurgery (SRS) treatment performed in two fractions at three-month intervals and post-procedural period were uneventful. Eight months later the patient was admitted to our hospital with headache, vomiting, right-sided facial palsy and right upper extremity paresthesia. Radiological examination demonstrated severe vasogenic edema in the left centrum semiovale and temporal region. Due to severe and steroid-resistant malign edema, hyperbaric oxygen (HBO2) therapy was performed as an alternative treatment option. Neurological symptoms resolved completely after HBO2. Radiological examination demonstrated serious improvement of brain edema and mass effect.